| Literature DB >> 22922421 |
Georgia Malamut1, Bertrand Meresse, Virginie Verkarre, Sophie Kaltenbach, Nicolas Montcuquet, Jean-Paul Duong Van Huyen, Céline Callens, Julien Lenglet, Gabriel Rahmi, Elia Samaha, Brigitte Ranque, Elizabeth Macintyre, Isabelle Radford-Weiss, Olivier Hermine, Nadine Cerf-Bensussan, Christophe Cellier.
Abstract
Large granular lymphocyte leukemia (LGL) is characterized by clonal expansion of CD3+ T cells or CD3(-) natural killer cells and frequently is associated with autoimmune diseases. We describe 2 patients with celiac disease who no longer responded to gluten-free diets after they developed T-cell LGL, with intestinal localization of malignant lymphocytes. Flow cytometry phenotyping of isolated intestinal intraepithelial and lamina propria cells eliminated type II refractory celiac disease, identifying large-sized CD8(+)CD57(+) T cells. Treatment with a combination of cyclosporine and methotrexate restored the patients' sensitivity to gluten-free diets. LGL therefore might be a cause of refractory celiac disease that is sensitive to immunosuppressive therapy.Entities:
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Year: 2012 PMID： 22922421 DOI： 10.1053/j.gastro.2012.08.028
Source DB: PubMed Journal: Gastroenterology ISSN： 0016-5085 Impact factor: 22.682